Brandt C A, Djernes B, Strømkjaer H, Petersen M B, Pedersen S, Hindkjaer J, Brinch-Iversen J, Bruun-Petersen G
Institute of Human Genetics, Aarhus University, Denmark.
J Med Genet. 1994 Feb;31(2):99-102. doi: 10.1136/jmg.31.2.99.
We report on a newborn white male infant with marked dysmorphic features and various congenital malformations. The initial clinical evaluation showed Crouzon-like features as well as some features of trisomy 18 syndrome and trisomy 13 syndrome. The results from conventional cytogenetic analysis showed a structurally abnormal chromosome replacing one normal chromosome 18, but only by applying molecular cytogenetic methods could the architecture of this abnormal chromosome be characterised clearly. The primed in situ labelling (PRINS) technique, using a newly synthesised alpha 18 oligonucleotide, showed the dicentric pattern and direct chromosome painting established the origin to be from chromosome 18. The combination of conventional cytogenetics and molecular cytogenetics showed the karyotype in the proband to be 45,XY,-14,-18,-21,+t(14;21),+psu dic(18) (qter-->cen-->p11.3: :p11.3-->psu cen-->qter). This was supported by molecular analysis using chromosome 18 specific DNA markers, which showed the paternal origin of the abnormal chromosome.
我们报告了一名患有明显畸形特征和多种先天性畸形的白人男婴。最初的临床评估显示出类似克鲁宗综合征的特征以及18三体综合征和13三体综合征的一些特征。传统细胞遗传学分析结果显示,一条结构异常的染色体替代了一条正常的18号染色体,但只有应用分子细胞遗传学方法才能清楚地确定这条异常染色体的结构。使用新合成的α18寡核苷酸的引物原位标记(PRINS)技术显示出双着丝粒模式,直接染色体涂染确定其起源于18号染色体。传统细胞遗传学和分子细胞遗传学相结合显示,先证者的核型为45,XY,-14,-18,-21,+t(14;21),+psu dic(18) (qter-->cen-->p11.3::p11.3-->psu cen-->qter)。使用18号染色体特异性DNA标记的分子分析支持了这一结果,该分析显示异常染色体来自父方。